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Expert Opinion

Burning Mouth Syndrome

Case History Submitted by Randolph W. Evans, MD Expert Opinion by Lisa A. Drage, MD

Burning mouth syndrome (BMS) is a common dis- cosal cause mouth pain; a few common exam- order that is not well known to many neurologists and ples include , , aphthous specialists. , and . A thorough oral examina- tion to exclude these and other oral must be CLINICAL HISTORY completed prior to making this diagnosis. A 49-year-old female was referred by her primary The pain of BMS may be described as a burning, care physician with a one-and-a-half-year history of scalded, sore, “hot,” tingling, or numb sensation and daily constant burning or numbness of the entirety of occur anywhere in the oral cavity although most of- her and the back of her throat. She also com- ten on the anterior two thirds and tip of the tongue. plains that the inside of her mouth is sensitive. She The pain of BMS is often qualitatively compared to has had a dry mouth for the last year. She had seen an a . With a prevalence in the general popu- ENT physician, gastroenterologist, and dentist. Artifi- lation of around 3.7%,1 BMS most commonly affects cial saliva has not been helpful. She has tried a variety middle-aged and elderly women and has an average of pain pills without any help. She tried Mycostatin at duration of 2 to 3 years.2 the onset without any benefit. She has been treated Common conditions associated with mouth pain with triamcinolone dental paste without any benefit. included psychiatric disease (, , ob- She does not have any . sessive compulsive disorder, somatoform disorder, There is a past medical history of hyperlipidemia and cancerphobia), xerostomia (drug-related, connec- on colesevelam (Welchol) and mild depression on tive tissue disease [CTD], age-related), nutritional de- buproprion (Wellbutrin). ficiencies (B12, iron, , , B6), and allergic con- Oropharyngeal and neurological examinations tact stomatitis (specifically to flavorings and food addi- were normal. tives). Less common causes of mouth pain include geo- Serum zinc, ferritin, and levels graphic tongue, , denture-related pain, oral were normal. Complete blood count and glycosy- habits, denture sore mouth, diabetes, thyroid abnor- lated hemoglobin was normal. Sjogren were malities, and . Angiotensin-converting en- negative. zyme inhibitors are one specific that causes Questions.—What is the diagnosis? Would addi- mouth pain, but many cause xerostomia tional testing be of any benefit? What treatment would and may lead to mouth pain. Unusual neurological you recommend? causes of BMS that have been reported include pain EXPERT OPINION referred from tonsils or teeth, lingual nerve neuropa- BMS specifically refers to mouth pain in a patient thy, glossopharyngeal neuropathy, and acoustic neu- 3 who has a normal oral examination. Many oral mu- roma. Damage to the cranial nerves associated with taste may lead to BMS.4 To complicate matters, up to

Address correspondence to Dr. Lisa A. Drage, Department of 37% of patients may have more than one factor con- Dermatology, Mayo Clinic, 200 First St. S.W., Rochester, MN tributing to the mouth pain that must be identified and 55905. treated.2

1079 1080 September 2005

The evaluation for BMS should include an oral tricyclic , low-dose benzodiazepines or examination concentrating on signs of , can- doxepin, topical clonazepam or gabapentin are op- didiasis, xerostomia, or mucosal abnormalities and tions that have been successful.3,4 A recent placebo- a directed history. Once assured there are no oral controlled study reported efficacy from sucking a 1 mucosal diseases that could cause symptoms, the mg clonazepam tablet, holding the saliva near the pain identification and treatment of all correctable causes sites in the mouth without swallowing for 3 minutes, of mouth pain should be pursued. All medications and then spitting three times a day.6 should be reviewed for their tendency to cause xe- The patient described here must first receive a rostomia. Direct questions regarding depression, anx- thorough oral examination to be correctly termed a iety, and fear should be broached. Mouth BMS patient. Even though multiple physicians have care routines and exacerbations by food or oral care previously examined her, a middle-aged woman with preparations should be assessed. A history of pain mouth pain may be labeled as BMS when she actually associated with dental work, denture institution or has lichen planus or another primary mucosal disease. parafunctional behaviors (, tongue thrusting, This patient has had an appropriate evalua- clenching) should be documented. Further consulta- tion although further questioning about food additives tion and evaluation by psychiatry, dentistry, neurol- and oral care habits may lead me to patch testing to ogy, and ENT should be sought if indicated by initial assess for allergic . evaluation. Given this patient’s history of depression, further Laboratory evaluation may include evaluation for assessment by a psychiatrist to ensure her mood dis- nutritional deficiencies (Complete blood count [CBC], order is being optimally managed would be my next iron studies, B12, B6, folate, zinc) as well as fasting glu- step. In addition, since buproprion (Wellbutrin) is as- cose, glycosylated hemoglobin, and thyroid studies. If sociated with significant xerostomia (up to 64% of pa- suspected, a culture for candidiasis should be obtained. tients) I would request a trial of an alternative antide- Patch testing (including oral flavors and preservatives) pressant with less potential for xerostomia to see if the may be a helpful adjunct. Since BMS is, by definition, buproprion is the cause of her burning mouth symp- associated with a normal oral examination, a biopsy is toms. Obviously,if the institution of her neither needed nor helpful. coincided with her BMS onset, this would heighten Treatment should be based on the results of this di- my suspicion. Colesevelam (Welchol) is reported to rected history, oral examination, and laboratory evalu- cause sore throat pain and should be discontinued for ation and should be tailored to the suspected causes of 4to6weeks or replaced to ensure that this is not the mouth pain in the specific patient.2,5 Some examples culprit. of tailored treatment include avoidance of irritants If optimal management of her depression on an such as alcohol-based mouth wash and flavored dental alternative medication and discontinuation of cole- product; avoidance of documented on patch sevelam does not improve the situation, low-dose testing; discontinuation of angiotensin-converting en- amitriptyline, sucking on clonazepam tablets as above zyme (ACE) inhibitors or medications that cause xe- or alternatively gabapentin to help control her symp- rostomia, denture evaluation, and adaptation; replace- toms could be instituted. ment of nutritional deficiencies, antiyeast agents; and evaluation and treatment of underlying psychiatric REFERENCES disease. 1. Bergdahl M, Bergdahl J. Burning mouth syndrome: If no cause of mouth pain is identified or the ini- prevalence and associated factors. JOral Pathol Med. tial treatment protocol is unsuccessful then use of a 1999;28:350-354. chronic pain algorithm would be appropriate, similar 2. Drage LA, Rogers RS. Clinical assessment and out- to that used for the medical management of neuro- come in 70 patients with complaints of burning or sore pathic pain conditions. Treatment with low doses of moth symptoms. Mayo Clin Proc. 1999;74:223-228. Headache 1081

3. Drage LA, Rogers RS. Burning mouth syndrome. Der- logical factors in burning mouth syndrome. BMJ. matol Clin. 2003;21:135-145. 1988;296:1243-1246. 4. Grushka M. Burning mouth syndrome. Am Fam Physi- 6. Gremeau-Richard C, Woda A, Navez ML, et al. cian. 2002;65:615-620. Topical clonazepam in stomatodynia: a randomised 5. Lamey PJ, Lamb AB. Prospective study of aetio- placebo-controlled study. Pain. 2004;108:51-57.